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A Phase I/II Study of BMN053 in Subjects With Duchenne Muscular Dystrophy (DMD)

6 december 2017 bijgewerkt door: BioMarin Pharmaceutical

A Phase I/II, Open-label, Dose Escalating With 48 Week Treatment Study to Assess the Safety and Tolerability, Pharmacokinetics, Pharmacodynamics and Efficacy of BMN053 (Previously Known as PRO053) in Subjects With Duchenne Muscular Dystrophy.

The purpose of the study is to see whether BMN053 is safe and effective to use as medication for Duchenne muscular dystrophy (DMD) patients with a mutation around location 53 in the DNA for the dystrophin protein.

Studie Overzicht

Gedetailleerde beschrijving

A Phase I/II, open-label, dose escalating with 48-week treatment study to assess the safety and tolerability, pharmacokinetics, pharmacodynamics and efficacy of BMN 053 (previously known as PRO053) in subjects with Duchenne muscular dystrophy

Studietype

Ingrijpend

Inschrijving (Werkelijk)

9

Fase

  • Fase 2
  • Fase 1

Contacten en locaties

In dit gedeelte vindt u de contactgegevens van degenen die het onderzoek uitvoeren en informatie over waar dit onderzoek wordt uitgevoerd.

Studie Locaties

      • Leuven, België, 3000
        • UZ Leuven, Campus Gasthuisberg
      • Paris, Frankrijk, 75651
        • Institut de Myologie
      • Rome, Italië, 00168
        • Policlinico Universitario Agostino Gemelli
      • Leiden, Nederland, 2333ZA
        • Leids Universitair Medisch Centrum
      • London, Verenigd Koninkrijk, WC1N 3JH
        • Great Ormond Street Hospital for Children
      • Newcastle, Verenigd Koninkrijk, NE1 3BZ
        • Institute of Genetic Medicine International Centre for Life

Deelname Criteria

Onderzoekers zoeken naar mensen die aan een bepaalde beschrijving voldoen, de zogenaamde geschiktheidscriteria. Enkele voorbeelden van deze criteria zijn iemands algemene gezondheidstoestand of eerdere behandelingen.

Geschiktheidscriteria

Leeftijden die in aanmerking komen voor studie

5 jaar tot 18 jaar (Kind, Volwassen)

Accepteert gezonde vrijwilligers

Nee

Geslachten die in aanmerking komen voor studie

Mannelijk

Beschrijving

Inclusion Criteria:

  1. Duchenne muscular dystrophy resulting from a mutation correctable by treatment with BMN053 confirmed by a state-of-the-art DNA diagnostic technique covering all DMD gene exons, including but not limited to MLPA (Multiplex Ligation-dependent Probe Amplification), CGH (Comparative Genomic Hybridisation), SCAIP (Single Condition Amplification/Internal Primer) or HRMCA (High-Resolution Melting Curve Analysis).
  2. Ambulant boys aged at least 5 years on the day of first dosing able to walk for at least 300 metres in the 6 minute walking distance (6MWD) test. In addition, results of the 6MWD test must be within ±30 metres of each other at 2 of 3 pre-treatment visits (screen 1, 2 and baseline) prior to first BMN053 administration.
  3. Adequate quality for biopsy (confirmed with MRI) of the lateral head of the gastrocnemius muscle. Only under exceptional circumstances will an alternative muscle (preferably brachii) be considered for biopsy and only following discussion between the Principal Investigator and the BioMarin Medical Monitor.
  4. Life expectancy of at least 3 years after inclusion in the study.
  5. Glucocorticosteroid use which is stable for at least 3 months prior to first BMN053 administration. Subjects must have been receiving glucocorticosteroids for at least 6 months prior to the first BMN053 administration.
  6. Willing and able to adhere to the study visit schedule and other protocol requirements.
  7. Written informed consent signed (by parent(s)/legal guardian and/or the subject, according to the local regulations).
  8. In France, a subject will be eligible for inclusion in this study only if either affiliated to, or a beneficiary of, a social security category.
  9. Anticipated adequate vein access for intravenous (IV) infusion.

Exclusion Criteria:

  1. Current or history of liver disease or impairment.
  2. Current or history of renal disease or impairment.
  3. At least two aPTT above upper limit of normal (ULN) within the last month prior to first dose of BMN053.
  4. Screening platelet count below the lower limit of normal (LLN).
  5. Acute illness within 4 weeks prior to first dose of BMN053 which may interfere with the study assessments.
  6. Severe mental retardation and/or behavioural problems which, in the opinion of the Investigator, prohibit participation in this study.
  7. Severe cardiomyopathy which, in the opinion of the Investigator prohibits participation in this study. If a subject has a left ventricular ejection fraction <45% at screening, the Investigator should discuss inclusion of the subject with the Medical Monitor.
  8. Expected need for daytime mechanical ventilation within the next year.
  9. Use of anticoagulants, antithrombotics or antiplatelet agents.
  10. Use of idebenone or other forms of coenzyme Q10 within 1 month prior to the start of the screening for the study.
  11. Use of nutritional or herbal supplements which, in the opinion of the Investigator, may influence muscle performance within 1 month prior to first dose of BMN053.
  12. Use of any other investigational product or participation in another trial with an investigational product, within 6 months prior to the start of the screening for the study.

Studie plan

Dit gedeelte bevat details van het studieplan, inclusief hoe de studie is opgezet en wat de studie meet.

Hoe is de studie opgezet?

Ontwerpdetails

  • Primair doel: Behandeling
  • Toewijzing: Niet-gerandomiseerd
  • Interventioneel model: Parallelle opdracht
  • Masker: Geen (open label)

Wapens en interventies

Deelnemersgroep / Arm
Interventie / Behandeling
Experimenteel: Dose escalation phase
In the dose-escalation phase, following screening assessment, two cohorts of three subjects each receive two single doses of BMN 053 in two study periods (i.e., four single doses in total per subject). In each study period they will receive BMN 053 by IV infusion and by SC injection (separated by one week). The proposed doses are 1 mg/kg (Cohort 1, study period 1), 3 mg/kg (Cohort 2, study period 1), 6 mg/kg (Cohort 1, study period 2) and 9 mg/kg (Cohort 2, study period 2). The actual doses may be amended or repeated based on emerging data from previous doses.
All doses of BMN053 have been administered as subcutaneous injections.
Experimenteel: Regimen selection
After completion of the dose-escalation period of Cohort 1, the safety data of the subjects will be reviewed by a DSMB and if no safety concerns these subjects will continue to receive 6 mg/kg BMN053 weekly by SC injection for 48 weeks. 3 more treatment-naïve subjects will be entered into this Group. These 6 subjects will form Group 1 of the Regimen Selection phase who received 6 mg/kg SC. At the time of this amendment (4) this part of the study has been completed. Following completion of the dose-escalation study period of Cohort 2 (9 mg/kg), the planned review of the preliminary plasma PK data from the dose-escalation phase showed a relative bioavailability of 50% for BMN053 with SC dosing (50% lower plasma AUC after SC dosing compared to IV dosing). Taking into consideration the risk of injection site reactions noted with similar compounds when administered SC over longer term, the planned 9 mg/kg BMN053 weekly by SC injection will be discontinued to be replaced by an IV regimen.

All doses of BMN053 will be administered as IV infusions. The proposed doses are as follows:

• 3 mg/kg

All doses of BMN053 will be administered as IV infusions. The proposed doses are as follows:

• 4-6 mg/kg

Experimenteel: 48-week Treatment Phase

Thirty additional treatment-naïve subjects will be recruited for the primary evaluation and will receive treatment at the recommended regimen for a total of 48 weeks. Subjects dosed initially in the dose escalation phase and/or the regimen selection phase of the study will not be included in the primary analysis.

Following completion of the 2nd study period for Cohort 2, the safety data will be reviewed by the DSMB and in the absence of safety concerns the subjects may enter the 48 week treatment phase and receive 9 mg/kg PRO053 once weekly by SC injection. Three new subjects will enter cohort 2 (i.e. 6 subjects in total at this dose level).

After the initial 12 subjects have completed 12 weeks of dosing the dose for the Treatment group (30 new subjects) will be selected based on the totality of the 12-week data from those initial 12 subjects. The initial 12 subjects will also be dosed on the selected dose (i.e. continue on their dose or [down-]titrate).

All doses of PRO053 will be administered as IV infusions. The proposed doses will be decided upon completion of the Regimen Selection Phase of Groups 2 and 3
Experimenteel: Dosing extension
All subjects who have completed the dose escalation and regimen selection phase of the study (N=15), and subjects who have complete the treatment phase of the study who have tolerated the treatment will be offered to continue dosing in the dosing extension with ongoing assessment of efficacy, safety, and tolerability of BMN 053. Safety, efficacy, PK/PD and biomarker assessments will be performed at scheduled visits; adverse events (AEs) and concomitant medications and therapies will be continuously monitored. The dose extension phase will provide BMN 053 treatment for 48 weeks.
All doses of PRO053 will be administered as IV infusions. The proposed doses will be decided upon completion of the Regimen Selection Phase of Groups 2 and 3 and the Treatment Phase Group 4.

Wat meet het onderzoek?

Primaire uitkomstmaten

Uitkomstmaat
Tijdsspanne
Verandering ten opzichte van de uitgangswaarde in een looptest van 6 minuten
Tijdsspanne: na 48 weken behandelingsfase
na 48 weken behandelingsfase

Secundaire uitkomstmaten

Uitkomstmaat
Tijdsspanne
Spierkracht
Tijdsspanne: na een behandelingsfase van 48 weken
na een behandelingsfase van 48 weken
Muscle function
Tijdsspanne: after 48 weeks treatment phase
after 48 weeks treatment phase
Pulmonary function
Tijdsspanne: after 48 weeks treatment phase
after 48 weeks treatment phase
Functional outcomes questionnaire
Tijdsspanne: after 48 weeks treatment phase
after 48 weeks treatment phase
Adverse Events
Tijdsspanne: after single intravenous and subcutaneous doses, and after 48 weeks of treatment phase
after single intravenous and subcutaneous doses, and after 48 weeks of treatment phase
Safety Laboratory
Tijdsspanne: after single intravenous and subcutaneous doses, and after 48 weeks of treatment phase
after single intravenous and subcutaneous doses, and after 48 weeks of treatment phase
Cardiac function
Tijdsspanne: after single intravenous and subcutaneous doses, and after 48 weeks of treatment phase
after single intravenous and subcutaneous doses, and after 48 weeks of treatment phase
Pharmacokinetic parameters at different dose levels
Tijdsspanne: after single intravenous and subcutaneous doses, and after 48 weeks of treatment phase
after single intravenous and subcutaneous doses, and after 48 weeks of treatment phase
Presence of (BMD-like) dystrophin expression in muscle biopsy
Tijdsspanne: after 48 weeks treatment phase
after 48 weeks treatment phase
Production of exon skip 53 mRNA in muscle biopsy
Tijdsspanne: after 48 weeks treatment phase
after 48 weeks treatment phase

Medewerkers en onderzoekers

Hier vindt u mensen en organisaties die betrokken zijn bij dit onderzoek.

Onderzoekers

  • Hoofdonderzoeker: V. Straub, Prof., Institute of Genetic Medicine, Newcastle University, UK

Publicaties en nuttige links

De persoon die verantwoordelijk is voor het invoeren van informatie over het onderzoek stelt deze publicaties vrijwillig ter beschikking. Dit kan gaan over alles wat met het onderzoek te maken heeft.

Nuttige links

Studie record data

Deze datums volgen de voortgang van het onderzoeksdossier en de samenvatting van de ingediende resultaten bij ClinicalTrials.gov. Studieverslagen en gerapporteerde resultaten worden beoordeeld door de National Library of Medicine (NLM) om er zeker van te zijn dat ze voldoen aan specifieke kwaliteitscontrolenormen voordat ze op de openbare website worden geplaatst.

Bestudeer belangrijke data

Studie start (Werkelijk)

1 juni 2013

Primaire voltooiing (Werkelijk)

3 augustus 2016

Studie voltooiing (Werkelijk)

3 augustus 2016

Studieregistratiedata

Eerst ingediend

2 juli 2013

Eerst ingediend dat voldeed aan de QC-criteria

7 oktober 2013

Eerst geplaatst (Schatting)

8 oktober 2013

Updates van studierecords

Laatste update geplaatst (Werkelijk)

8 december 2017

Laatste update ingediend die voldeed aan QC-criteria

6 december 2017

Laatst geverifieerd

1 december 2017

Meer informatie

Deze informatie is zonder wijzigingen rechtstreeks van de website clinicaltrials.gov gehaald. Als u verzoeken heeft om uw onderzoeksgegevens te wijzigen, te verwijderen of bij te werken, neem dan contact op met register@clinicaltrials.gov. Zodra er een wijziging wordt doorgevoerd op clinicaltrials.gov, wordt deze ook automatisch bijgewerkt op onze website .

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